Literature DB >> 23315038

Friedreich ataxia: dysarthria profile and clinical data.

Bettina Brendel1, Hermann Ackermann, Daniela Berg, Tobias Lindig, Theresa Schölderle, Ludger Schöls, Matthis Synofzik, Wolfram Ziegler.   

Abstract

Friedreich ataxia (FRDA) is the most frequent recessive ataxia in the Western world. Dysarthria is a cardinal feature of FRDA, often leading to severe impairments in daily functioning, but its exact characteristics are only poorly understood so far. We performed a comprehensive evaluation of dysarthria severity and the profile of speech motor deficits in 20 patients with a genetic diagnosis of FRDA based on a carefully selected battery of speaking tasks and two widely used paraspeech tasks, i.e., oral diadochokinesis and sustained vowel productions. Perceptual ratings of the speech samples identified respiration, voice quality, voice instability, articulation, and tempo as the most affected speech dimensions. Whereas vocal instability predicted ataxia severity, tempo turned out as a significant correlate of disease duration. Furthermore, articulation predicted the overall intelligibility score as determined by a systematic speech pathology assessment tool. In contrast, neurologists' ratings of intelligibility--a component of the "Scale for the Assessment and Rating of Ataxia"--were found to be related to perceived speech tempo. Obviously, clinicians are more sensitive to slowness of speech than to any other feature of spoken language during dysarthria evaluation. Our results suggest that different components of speech production and trunk/limb motor functions are differentially susceptible to FRDA pathology. Furthermore, evidence emerged that paraspeech tasks do not allow for an adequate scaling of speech deficits in FRDA.

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Year:  2013        PMID: 23315038     DOI: 10.1007/s12311-012-0440-0

Source DB:  PubMed          Journal:  Cerebellum        ISSN: 1473-4222            Impact factor:   3.847


  31 in total

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3.  Task-related factors in oral motor control: speech and oral diadochokinesis in dysarthria and apraxia of speech.

Authors:  Wolfram Ziegler
Journal:  Brain Lang       Date:  2002-03       Impact factor: 2.381

Review 4.  Friedreich ataxia: the clinical picture.

Authors:  Massimo Pandolfo
Journal:  J Neurol       Date:  2009-03       Impact factor: 4.849

5.  Telediagnostic assessment of intelligibility in dysarthria: a pilot investigation of MVP-online.

Authors:  Wolfram Ziegler; Andreas Zierdt
Journal:  J Commun Disord       Date:  2008-05-16       Impact factor: 2.288

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Authors:  Franco Taroni; Stefano DiDonato
Journal:  Nat Rev Neurosci       Date:  2004-08       Impact factor: 34.870

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Authors:  H Ackermann; W Ziegler
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Journal:  Folia Phoniatr Logop       Date:  1995       Impact factor: 0.849

Review 9.  Diagnosis and treatment of Friedreich ataxia: a European perspective.

Authors:  Jörg B Schulz; Sylvia Boesch; Katrin Bürk; Alexandra Dürr; Paola Giunti; Caterina Mariotti; Francoise Pousset; Ludger Schöls; Pierre Vankan; Massimo Pandolfo
Journal:  Nat Rev Neurol       Date:  2009-04       Impact factor: 42.937

10.  Brain structural damage in Friedreich's ataxia.

Authors:  R Della Nave; A Ginestroni; M Giannelli; C Tessa; E Salvatore; F Salvi; M T Dotti; G De Michele; S Piacentini; M Mascalchi
Journal:  J Neurol Neurosurg Psychiatry       Date:  2007-07-18       Impact factor: 10.154

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  9 in total

Review 1.  Consensus Paper: Neurophysiological Assessments of Ataxias in Daily Practice.

Authors:  W Ilg; M Branscheidt; A Butala; P Celnik; L de Paola; F B Horak; L Schöls; H A G Teive; A P Vogel; D S Zee; D Timmann
Journal:  Cerebellum       Date:  2018-10       Impact factor: 3.847

2.  Comparing speech characteristics in spinocerebellar ataxias type 3 and type 6 with Friedreich ataxia.

Authors:  Bettina Brendel; Matthis Synofzik; Hermann Ackermann; Tobias Lindig; Theresa Schölderle; Ludger Schöls; Wolfram Ziegler
Journal:  J Neurol       Date:  2014-09-30       Impact factor: 4.849

3.  Adult normative values for the PATA Rate Test.

Authors:  Chiara Pane; Teresa Costabile; Adriana Salvati; Dalila Luisa Aurisicchio; Filomena Abate; Angese Liguori; Francesca Paciello; Silvio Peluso; Fiore Manganelli; Giuseppe De Michele; Alessandro Filla; Francesco Saccà
Journal:  J Neurol       Date:  2018-03-06       Impact factor: 4.849

Review 4.  Nonspeech Oral Movements and Oral Motor Disorders: A Narrative Review.

Authors:  Ray D Kent
Journal:  Am J Speech Lang Pathol       Date:  2015-11       Impact factor: 2.408

Review 5.  Congenital or Early Developing Neuromuscular Diseases Affecting Feeding, Swallowing and Speech - A Review of the Literature from January 1998 to August 2021.

Authors:  Lotta Sjögreen; Lisa Bengtsson
Journal:  J Neuromuscul Dis       Date:  2022

6.  Dysphagia and swallowing-related quality of life in Friedreich ataxia.

Authors:  Adam P Vogel; Sophie E Brown; Joanne E Folker; Louise A Corben; Martin B Delatycki
Journal:  J Neurol       Date:  2013-12-27       Impact factor: 4.849

7.  Dimethyl fumarate dose-dependently increases mitochondrial gene expression and function in muscle and brain of Friedreich's ataxia model mice.

Authors:  Chun Kiu Hui; Elena N Dedkova; Claire Montgomery; Gino Cortopassi
Journal:  Hum Mol Genet       Date:  2021-02-25       Impact factor: 5.121

8.  Feasibility and Acceptability of Lee Silverman Voice Treatment in Progressive Ataxias.

Authors:  Anja Lowit; Aisling Egan; Marios Hadjivassiliou
Journal:  Cerebellum       Date:  2020-10       Impact factor: 3.847

9.  ClearSpeechTogether: a Rater Blinded, Single, Controlled Feasibility Study of Speech Intervention for People with Progressive Ataxia.

Authors:  Anja Lowit; Jessica Cox; Melissa Loucas; Jennifer Grassly; Aisling Egan; Frits van Brenk; Marios Hadjivassiliou
Journal:  Cerebellum       Date:  2022-08-24       Impact factor: 3.648

  9 in total

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